Outline

Introduction: Clinical relevance of antibody-mediated rejection (AMR) in vascularized composite allotransplantation (VCA) has not been defined. While C4d deposition and DSAs have been previously described in hand and face transplant recipients, no clinical relevance has been reported. We herein describe a novel type of DSA and B-cell associated rejection in hand transplantation.

Material and methods: A 41-year-old male had lost two thirds of his forearms in an electric current accident in 2000 and received bilateral forearm transplantation in 2003. Six previous rejections required treatment with steroids, basiliximab, ATG, alemtuzumab or tacrolimus dose augmentation. DSAs have remained negative during this course. In February 2012, the patient presented with edematous hands and forearms without any exanthema or efflorescences.

Results: Punch-skin biopsies revealed rejection grade Banff II. Immunhistochemical analysis identified large aggregates of lymphocytes with an architecture resembling lymph nodes. As per CD20 staining, the center oft he aggregates almost entirely consisted of B-lymphocytes. DSAs (Luminex) were found at a high level. The patient was first treated with steroids and an increase of maintenance immunosuppression but did not respond to this treatment. Based on the predominance of B-cells and DSAs with lack of response to conventional, Rituximab-therapy was given at 375 mg/m2 BSA. The severe edema of the transplanted forearms disappeared within one month. Control biopsy showed normal skin with absence of B-cells. DSAs were negative at 3 months after rituximab.

Conclusion: We herein report the first case of what we suggest is an AMR in VCA occuring at nine years after forearm transplantation, Rituximab therapy successfully reversed the event.